Cargando…

Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study

Temper outbursts are a severe problem for people with Prader-Willi Syndrome (PWS). Previous reports indicate that vagus nerve stimulation (VNS) may reduce maladaptive behaviour in neurodevelopmental disorders, including PWS. We systematically investigated the effectiveness of transcutaneous VNS (t-V...

Descripción completa

Detalles Bibliográficos
Autores principales: Manning, Katherine E., Beresford-Webb, Jessica A., Aman, Lucie C. S., Ring, Howard A., Watson, Peter C., Porges, Stephen W., Oliver, Chris, Jennings, Sally R., Holland, Anthony J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6890246/
https://www.ncbi.nlm.nih.gov/pubmed/31794560
http://dx.doi.org/10.1371/journal.pone.0223750
_version_ 1783475571576012800
author Manning, Katherine E.
Beresford-Webb, Jessica A.
Aman, Lucie C. S.
Ring, Howard A.
Watson, Peter C.
Porges, Stephen W.
Oliver, Chris
Jennings, Sally R.
Holland, Anthony J.
author_facet Manning, Katherine E.
Beresford-Webb, Jessica A.
Aman, Lucie C. S.
Ring, Howard A.
Watson, Peter C.
Porges, Stephen W.
Oliver, Chris
Jennings, Sally R.
Holland, Anthony J.
author_sort Manning, Katherine E.
collection PubMed
description Temper outbursts are a severe problem for people with Prader-Willi Syndrome (PWS). Previous reports indicate that vagus nerve stimulation (VNS) may reduce maladaptive behaviour in neurodevelopmental disorders, including PWS. We systematically investigated the effectiveness of transcutaneous VNS (t-VNS) in PWS. Using a non-blind single case repeat measures modified ABA design, with participants as their own controls, t-VNS was evaluated in five individuals with PWS [three males; age 22–41 (M = 26.8)]. After a baseline phase, participants received four-hours of t-VNS daily for 12 months, followed by one month of daily t-VNS for two-hours. The primary outcome measure was the mean number of behavioural outbursts per day. Secondary outcomes included findings from behavioural questionnaires and both qualitative and goal attainment interviews. Four of the five participants who completed the study exhibited a statistically significant reduction in number and severity of temper outbursts after approximately nine months of daily four-hour t-VNS. Subsequent two-hour daily t-VNS was associated with increased outbursts for all participants, two reaching significance. Questionnaire and interview data supported these findings, the latter indicating potential mechanisms of action. No serious safety issues were reported. t-VNS is an effective, novel and safe intervention for chronic temper outbursts in PWS. We propose these changes are mediated through vagal projections and their effects both centrally and on the functioning of the parasympathetic nervous system. These findings challenge our present biopsychosocial understanding of such behaviours suggesting that there is a single major mechanism that is modifiable using t-VNS. This intervention is potentially generalizable across other clinical groups. Future research should address the lack of a sham condition in this study along with the prevalence of high drop out rates, and the potential effects of different stimulation intensities, frequencies and pulse widths.
format Online
Article
Text
id pubmed-6890246
institution National Center for Biotechnology Information
language English
publishDate 2019
publisher Public Library of Science
record_format MEDLINE/PubMed
spelling pubmed-68902462019-12-13 Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study Manning, Katherine E. Beresford-Webb, Jessica A. Aman, Lucie C. S. Ring, Howard A. Watson, Peter C. Porges, Stephen W. Oliver, Chris Jennings, Sally R. Holland, Anthony J. PLoS One Research Article Temper outbursts are a severe problem for people with Prader-Willi Syndrome (PWS). Previous reports indicate that vagus nerve stimulation (VNS) may reduce maladaptive behaviour in neurodevelopmental disorders, including PWS. We systematically investigated the effectiveness of transcutaneous VNS (t-VNS) in PWS. Using a non-blind single case repeat measures modified ABA design, with participants as their own controls, t-VNS was evaluated in five individuals with PWS [three males; age 22–41 (M = 26.8)]. After a baseline phase, participants received four-hours of t-VNS daily for 12 months, followed by one month of daily t-VNS for two-hours. The primary outcome measure was the mean number of behavioural outbursts per day. Secondary outcomes included findings from behavioural questionnaires and both qualitative and goal attainment interviews. Four of the five participants who completed the study exhibited a statistically significant reduction in number and severity of temper outbursts after approximately nine months of daily four-hour t-VNS. Subsequent two-hour daily t-VNS was associated with increased outbursts for all participants, two reaching significance. Questionnaire and interview data supported these findings, the latter indicating potential mechanisms of action. No serious safety issues were reported. t-VNS is an effective, novel and safe intervention for chronic temper outbursts in PWS. We propose these changes are mediated through vagal projections and their effects both centrally and on the functioning of the parasympathetic nervous system. These findings challenge our present biopsychosocial understanding of such behaviours suggesting that there is a single major mechanism that is modifiable using t-VNS. This intervention is potentially generalizable across other clinical groups. Future research should address the lack of a sham condition in this study along with the prevalence of high drop out rates, and the potential effects of different stimulation intensities, frequencies and pulse widths. Public Library of Science 2019-12-03 /pmc/articles/PMC6890246/ /pubmed/31794560 http://dx.doi.org/10.1371/journal.pone.0223750 Text en © 2019 Manning et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Manning, Katherine E.
Beresford-Webb, Jessica A.
Aman, Lucie C. S.
Ring, Howard A.
Watson, Peter C.
Porges, Stephen W.
Oliver, Chris
Jennings, Sally R.
Holland, Anthony J.
Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title_full Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title_fullStr Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title_full_unstemmed Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title_short Transcutaneous vagus nerve stimulation (t-VNS): A novel effective treatment for temper outbursts in adults with Prader-Willi Syndrome indicated by results from a non-blind study
title_sort transcutaneous vagus nerve stimulation (t-vns): a novel effective treatment for temper outbursts in adults with prader-willi syndrome indicated by results from a non-blind study
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6890246/
https://www.ncbi.nlm.nih.gov/pubmed/31794560
http://dx.doi.org/10.1371/journal.pone.0223750
work_keys_str_mv AT manningkatherinee transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT beresfordwebbjessicaa transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT amanluciecs transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT ringhowarda transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT watsonpeterc transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT porgesstephenw transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT oliverchris transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT jenningssallyr transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy
AT hollandanthonyj transcutaneousvagusnervestimulationtvnsanoveleffectivetreatmentfortemperoutburstsinadultswithpraderwillisyndromeindicatedbyresultsfromanonblindstudy